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Isolated tubercular dacryoadenitis in a non-immunocompromised healthy adult


1 Department of Ophthalmology, Jawaharlal Postgraduate Medical Education and Research, Puducherry, India
2 Department of Pathology, Jawaharlal Postgraduate Medical Education and Research, Puducherry, India

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Date of Submission18-May-2021
Date of Decision27-Sep-2021
Date of Acceptance27-Sep-2021
Date of Web Publication07-Jun-2022
 

   Abstract 


A middle-aged woman presented with a slowly enlarging mass in the right superotemporal orbit and ptosis. The patient had an elevated erythrocyte sedimentation rate, and incisional biopsy showed granulomatous inflammation with Langhan's giant cells suggestive of a tubercular etiology. Tuberculin skin test was negative. The patient responded well to anti-tuberculous therapy. Tubercular dacryoadenitis is a rare presentation of a lacrimal gland mass.

Keywords: Lacrimal gland, orbital, tuberculosis


How to cite this URL:
Jossy A, Kaviyapriya N, Kasturi N, Rekha J S. Isolated tubercular dacryoadenitis in a non-immunocompromised healthy adult. Indian J Pathol Microbiol [Epub ahead of print] [cited 2022 Dec 9]. Available from: https://www.ijpmonline.org/preprintarticle.asp?id=346855





   Introduction Top


Tuberculosis (TB) is one of the leading causes of morbidity and mortality worldwide. The estimated TB prevalence in India for the year 2015 is about 2.5 million cases.[1] Extrapulmonary form of tuberculosis is seen in 15–25% of all cases of tuberculosis, of which ocular tuberculosis constitutes 2–30% of extrapulmonary cases; choroid involvement being the commonest and orbital or periorbital tuberculosis constituting a very small fraction of these cases.[2] Of the etiologies that induce enlargement of the lacrimal gland, approximately 50% are infectious or inflammatory, 25% are lymphoid tumors, and 25% are salivary tumors.[3] Tubercular dacryoadenitis is very rare but still makes an important differential diagnosis of lacrimal gland swellings, especially in endemic areas like India, with the diagnosis usually made based on histopathological examination of the lacrimal gland.[4]

The study adhered to the principles of the Declaration of Helsinki; the patient has consented to publish her identifiable photograph.


   Case Top


A 35-year-old Indian female presented with a swelling in the superolateral quadrant of the right orbit for the past 15 days [Figure 1]a and [Figure 1]b. The swelling was insidious in onset, painless, associated with minimal watering and redness of the right eye. She had no history of trauma, sinusitis, steroid intake, fever, diabetes, or any other significant medical history. On examination, her best-corrected visual acuity in both eyes was 20/20, N6. Examination of the right eye showed a 3 cm × 2 cm firm, tender palpable mass in the supertemporal quadrant of the orbit with severe ptosis. Extraocular movements were full. Slit-lamp examination of the right eye revealed conjunctival congestion, mild chemosis, and discharge with few papillae. The rest of the anterior segment and fundus examination was within normal limits. Examination of the left eye was unremarkable. General and systemic examinations were normal. Routine hematological investigations, chest X-ray, and retrovirus testing were normal, but her erythrocyte sedimentation rate (ESR) was found to be elevated (54 mm/hr). Tuberculin skin test showed 5 mm induration. Computed tomography (CT) scan of the orbits showed an irregular soft tissue lesion involving the right lacrimal gland, preseptal soft tissue space, with extension into the insertion of the right lateral rectus muscle and anterior portion of the lateral extraconal space [Figure 2]a. No calcification or bony erosion was noted. The absence of both, cystic content and normal sinuses ruled out the possibility of a ruptured dermoid cyst and an invasive fungal infection, respectively. Acute dacryoadenitis was suspected, and the patient was given a course of systemic and topical antibiotics for two weeks. The patient showed no signs of improvement on follow-up. An incision biopsy of the lesion was done, and histopathological examination showed fibro collagenous tissue with multiple well-formed coalescing granulomas composed of epithelioid histiocytes, few lymphocytes, and occasional Langhan's giant cells suggestive of granulomatous inflammation—probably tuberculous etiology [Figure 2]b. Acid-fast bacilli staining was negative. Serum calcium, angiotensin-converting enzyme, lysozyme levels, and C-ANCA were normal upon testing. A diagnosis of tubercular dacryoadenitis was therefore made, and the patient was started on standard anti-tuberculous therapy (ATT) for six months. The swelling decreased significantly within three weeks of starting treatment, however, the firm mass was still palpable [Figure 3]. The patient completed six months of anti-tubercular therapy with resolution of proptosis at the end of treatment.
Figure 1: (a and b) Clinical photograph showing the swelling in the right upper lid with ptosis

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Figure 2: (a) Axial MRI scan of the orbit showing enlarged lacrimal gland in the superotemporal orbit and (b) Histopathology image 400 × with Hematoxylin-eosin stain, showing tubercular granulomas

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Figure 3: (a and b) Clinical photograph showing a reduction in lacrimal gland swelling and ptosis following anti-tubercular treatment

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   Discussion Top


Dacryoadenitis is an inflammation of the lacrimal gland. The initial symptoms typically consist of discomfort in the region of the lacrimal gland (i.e., the supertemporal orbit), unilaterally or bilaterally, and occasionally with pain.[5] However, severe cases may be characterized by edema, erythema, and tenderness in the tissues surrounding these glands. Dacryoadenitis is a rare manifestation of tuberculosis. Other causes of lacrimal gland enlargement include lymphoma and sarcoid; however, these conditions are usually bilateral.[6] Very few cases of tubercular dacryoadenitis have been reported in the literature. Ocular tuberculosis involving the lacrimal gland was first described by Abadie in 1881. Since then, around 84 cases have been described in the literature and about 50 cases among them are from India.[7],[8] Lacrimal gland involvement usually occurs in association with periostitis or osteomyelitis, usually affecting the lateral orbital wall or with the involvement of the orbital soft tissue or an abscess.[9] Isolated lacrimal gland involvement has also been reported, which is usually of the sclerotic type and rarely of the caseous type.[7] In a series of 10,542 cases of tuberculosis, the incidence of ocular tuberculosis of 1.4%, but no cases of dacryoadenitis were encountered.[10] Females in endemic areas aged between 35 and 50 years are predominantly affected by this manifestation, presenting usually painless swelling of the eyelid, which may enlarge to form an abscess or a discharging sinus.[11] Acute dacryoadenitis that usually does not resolve with antibiotics within 3 to 4 weeks or present with atypical features like occurrence in older adults, bilateral presentation, and systemic symptoms may warrant additional workup. Diagnosis of ocular TB can be challenging, and the clinical findings mimic those of a wide array of different underlying diseases, necessitating a complete workup to exclude other causes leading to delays in diagnosis and initiation of ATT. Sarcoidosis and Wegener's granulomatosis were ruled out in our patient due to the absence of systemic signs and normal laboratory parameters. Chest x-ray or computerized tomography demonstrating cavities, consolidation, or lymph node enlargement should heighten suspicion for primary TB infection. However, it should be kept in mind that the absence of clinically evident pulmonary TB does not rule out the possibility of ocular TB, as about 60% of patients with extrapulmonary TB have no evidence of pulmonary TB.[12] For a definitive diagnosis, polymerase chain reaction and isolation of Mycobacterium tuberculosis and required, but it is rare to get a positive culture from the lacrimal gland secretions or a fine needle aspiration technique.[13] A biopsy is generally performed via a transcutaneous approach, sampling the orbital lobe to avoid the tear ducts that drain through the palpebral lobe to the temporal conjunctival fornix. Drug regimens for ocular tuberculosis are similar to those for pulmonary or extrapulmonary tuberculosis. The exact duration of treatment and the endpoint for stopping treatment for ocular tuberculosis is not known. However, in an endemic country like ours, a minimum duration of 9 to 12 months is recommended. Our patient had shown a marked response to ATT with a notable decrease in the symptoms and the size of the lesion. Proper compliance must be ensured to prevent drug resistance, which is of significant concern in an endemic country.

Tubercular dacryoadenitis, although rare, must be considered as one of the differentials in cases of dacryoadenitis not responding to conventional antibiotics or anti-inflammatory agents. Biopsy and response to anti-tubercular medications remain the mainstay of diagnosis.

Declaration of Patient Consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial Support and Sponsorship

Nil.

Conflicts of Interest

There are no conflicts of interest.



 
   References Top

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4.
Sharma A, Pandey SK, Mohan K, Khandelwal N, Gupta A. Tuberculosis of the lacrimal gland: A case report. J Pediatr Ophthalmol Strabismus 1998;35:237-9.  Back to cited text no. 4
    
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Hina K, Seema M, Taskin K, Mehvish H. Acute lacrimal gland swelling with intracranial extension and without any neurological features – A rare case report. Saudi J Ophthalmol 2018;32:253-6.  Back to cited text no. 5
    
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Narula MK, Chaudhary V, Baruah D, Kathuria M, Anand R. Pictorial essay: Orbital tuberculosis. Indian J Radiol Imaging 2010;20:6-10.  Back to cited text no. 6
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7.
Mortada A. Tuberculoma of the orbit and lacrimal gland. Br J Ophthalmol 1971;55:565-7.  Back to cited text no. 7
    
8.
Babu K, Mukhopadhyay M, Bhat SS, Chinmayee J. Orbital and adnexal tuberculosis: A case series from a South Indian population. J Ophthalmic Inflamm Infect 2014;4:12.  Back to cited text no. 8
    
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Chakraborti C, Biswas R, Mondal M, Mukhopadhya U, Datta J. Tuberculous dacryoadenitis in a child. Nepal J Ophthalmol 2011;3:210-3.  Back to cited text no. 9
    
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Donahue HC. Ophthalmologic experience in a tuberculosis sanatorium. Am J Ophthalmol 1967;64:742-8.  Back to cited text no. 10
    
11.
Bist SS, Luthra M, Agarwal V, Bharti B. Isolated lacrimal gland tuberculosis: A rare clinical entity in a young child. Online J Health Allied Scs 2016;15:12.  Back to cited text no. 11
    
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Amber K, Priyanka, Mahesh M. Recent changes in tuberculosis guidelines: What's new for ocular tuberculosis. Tr Ophtha Open Acc J 2019;2. TOOAJ.MS.ID.000130. DOI: 10.32474/TOOAJ.2019.02.000130.  Back to cited text no. 12
    
13.
Biswas J, Roy Chowdhury B, Krishna Kumar S, Lily Therese K, Madhavan HN. Detection of Mycobacterium tuberculosis by polymerase chain reaction in a case of orbital tuberculosis. Orbit 2001;20:69-74.  Back to cited text no. 13
    

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Correspondence Address:
Nirupama Kasturi,
Department of Ophthalmology, Jawaharlal Postgraduate Medical Education and Research, Puducherry - 605 006
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijpm.ijpm_489_21



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  [Figure 1], [Figure 2], [Figure 3]



 

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